PubTransformer

A site to transform Pubmed publications into these bibliographic reference formats: ADS, BibTeX, EndNote, ISI used by the Web of Knowledge, RIS, MEDLINE, Microsoft's Word 2007 XML.

A case report of lymphoid intestitial pneumonia in common variable immunodeficiency: Oligoclonal expansion of effector lymphocytes with preferential cytomegalovirus-specific immune response and lymphoproliferative disease promotion.

Abstract Lymphoid interstitial pneumonia (LIP) is a rare disease with lymphocytic infiltration of the alveolar interstitial and air spaces, sometimes classified as a clonal lymphoproliferative disease (LPD) with high prevalence in patients with immunodysregulation. Although association of mucosa-associated lymphoid tissue (MALT) lymphoma development with infectious agents has been well described, it is not so in the case of LIP. Attempts to demonstrate an infective cause by direct microbe detection have failed, but association with atypical specific immune response to opportunistic infectious agent has not been studied.
PMID
Related Publications

Pulmonary radiologic findings in common variable immunodeficiency: clinical and immunological correlations.

Tertiary lymphoid neogenesis is a component of pulmonary lymphoid hyperplasia in patients with common variable immunodeficiency.

Chronic cytomegalovirus infection that present specific clinical course--a case of a boy with common variable immunodeficiency.

Mono- and oligoclonal immunoglobulin anomalies in kidney transplant patients.

Oligoclonal T cell expansions in pulmonary lymphoproliferative disorders: demonstration of the frequent occurrence of oligoclonal T cells in human immunodeficiency virus-related lymphoid interstitial pneumonia.

Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start




PMID- 28591035
OWN - NLM
STAT- MEDLINE
DA  - 20170607
DCOM- 20170706
LR  - 20170706
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 23
DP  - 2017 Jun
TI  - A case report of lymphoid intestitial pneumonia in common variable
      immunodeficiency: Oligoclonal expansion of effector lymphocytes with preferential
      cytomegalovirus-specific immune response and lymphoproliferative disease
      promotion.
PG  - e7031
LID - 10.1097/MD.0000000000007031 [doi]
AB  - RATIONALE: Lymphoid interstitial pneumonia (LIP) is a rare disease with
      lymphocytic infiltration of the alveolar interstitial and air spaces, sometimes
      classified as a clonal lymphoproliferative disease (LPD) with high prevalence in 
      patients with immunodysregulation. Although association of mucosa-associated
      lymphoid tissue (MALT) lymphoma development with infectious agents has been well 
      described, it is not so in the case of LIP. Attempts to demonstrate an infective 
      cause by direct microbe detection have failed, but association with atypical
      specific immune response to opportunistic infectious agent has not been studied. 
      PATIENT CONCERNS AND DIAGNOSES: We performed clinical, biochemical, and
      immunologic analysis of patients LIP that arises primarily from the common
      variable immune deficiency (CVID) with normal immunoglobulin class M (IgM) level 
      and mild infectious course as a result of immunodysregulation. At the age of 13
      multiple nodules, areas of consolidation were observed and LIP was confirmed by
      histological examination. The progression of the disease with massive
      splenomegaly (17-->27 cm), lymphadenopathy soft tissue infiltration coincides
      with high standardized uptake value (SUV was 3.1-5.2), regulatory T cells
      decrease (CD4+25FoxP3+ level -0.02%, i.e., 8 cells per 100 muL), oligoclonal
      gammapathy: very high IgM (3340 mg/dL) and beta2-microglobulin (18.8 mg/L) level 
      observed 10 years later.Immune response polarization was observed in humoral and 
      cellular compartment -Th and Tc-dependent: 10.8% of lymphocytes are CD8high+CMV
      pp65-pentamer positive cells (Epstein-Barr virus-specific not observed). Specific
      immune response polarization correlates with negative immunofixation, light
      chains kappa/lambda = 2.84 and narrow, but non-monoclonal T cell receptor (TCR)/ 
      B cell receptor (BCR) repertoire. LESSONS: Taking everything into account, this
      case report shows that LIP is a consequence of immune-dysregulation in CVID, that
      is, Treg deficiency, narrow lymphocyte repertoire, and abnormal ability to
      respond to cytomegalovirus (CMV) antigens. It may be visualized by positron
      emission tomography (PET) and monitored by CMV-specific immune response,
      beta2-microglobulin level, and IgM paraproteinaemia, but not by immunofixation
      and kappa/lambda ratio.
FAU - Zdziarski, Przemyslaw
AU  - Zdziarski P
AD  - aDepartment of Clinical Immunology, Lower Silesian Center for Cellular
      Transplantation bL Hirszfeld Institute of Immunology and Experimental Therapy,
      Polish Academy of Sciences, Wroclaw cDepartment of Immunology, Poznan University 
      of Medical Sciences, Poznan, Poland.
FAU - Gamian, Andrzej
AU  - Gamian A
FAU - Dworacki, Grzegorz
AU  - Dworacki G
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 0 (Antigens, Viral)
RN  - 0 (Immunoglobulin M)
RN  - 0 (Oligoclonal Bands)
RN  - Lymphoid Interstitial Pneumonia
SB  - AIM
SB  - IM
MH  - Adolescent
MH  - Antigens, Viral/metabolism
MH  - Common Variable Immunodeficiency/*complications/immunology
MH  - Cytomegalovirus/immunology
MH  - Female
MH  - Humans
MH  - Immunoglobulin M/metabolism
MH  - Lung Diseases, Interstitial/*etiology/immunology
MH  - Lymphocytes/immunology
MH  - Lymphoproliferative Disorders/*complications/immunology
MH  - Oligoclonal Bands
MH  - Paraproteinemias/etiology/immunology
PMC - PMC5466213
EDAT- 2017/06/08 06:00
MHDA- 2017/07/07 06:00
CRDT- 2017/06/08 06:00
AID - 10.1097/MD.0000000000007031 [doi]
AID - 00005792-201706090-00011 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Jun;96(23):e7031. doi: 10.1097/MD.0000000000007031.